Michael P Hong1, Janna L Guilfoyle1, Lindsey N Mooney1, Logan K Wink2, Ernest V Pedapati2, Rebecca C Shaffer1, John A Sweeney3, Craig A Erickson4. 1. Cincinnati Children's Hospital Medical Center, United States. 2. Cincinnati Children's Hospital Medical Center, United States; University of Cincinnati, College of Medicine, United States. 3. University of Cincinnati, College of Medicine, United States. 4. Cincinnati Children's Hospital Medical Center, United States; University of Cincinnati, College of Medicine, United States; Indiana University School of Medicine, United States. Electronic address: Craig.erickson@cchmc.org.
Abstract
BACKGROUND: Angelman syndrome (AS) is a rare neurological disorder characterized by severe developmental disability, communication impairment, elevated seizure risk, and motor system abnormalities. AIMS: The aims of this study were to determine the feasibility of social scene eye tracking and pupillometry measures in individuals with AS and to compare the performance of AS participants to individuals with idiopathic Autism Spectrum Disorder (ASD) and typically developing controls (TDC). METHODS AND PROCEDURES: Individuals with AS and age- and gender- matched controls completed a social eye tracking paradigm. Neurobehavioral characterization of AS participants was completed via a battery of psychological testing and caregiver behavioral evaluations. OUTCOMES AND RESULTS: Eight of seventeen recruited AS participants completed the eye tracking paradigm. Compared to TDC, AS subjects demonstrated significantly less preference for social scenes than geometric shapes. Additionally, AS subjects showed less pupil dilation, compared to TDC, when viewing social scenes versus geometric shapes. There was no statistically significant difference found between AS and ASD subjects in either social eye tracking or pupillometry. CONCLUSIONS AND IMPLICATIONS: The use of eye tracking and pupillometry may represent an innovative measure for quantifying AS-associated impairments in social salience.
BACKGROUND:Angelman syndrome (AS) is a rare neurological disorder characterized by severe developmental disability, communication impairment, elevated seizure risk, and motor system abnormalities. AIMS: The aims of this study were to determine the feasibility of social scene eye tracking and pupillometry measures in individuals with AS and to compare the performance of AS participants to individuals with idiopathic Autism Spectrum Disorder (ASD) and typically developing controls (TDC). METHODS AND PROCEDURES: Individuals with AS and age- and gender- matched controls completed a social eye tracking paradigm. Neurobehavioral characterization of AS participants was completed via a battery of psychological testing and caregiver behavioral evaluations. OUTCOMES AND RESULTS: Eight of seventeen recruited AS participants completed the eye tracking paradigm. Compared to TDC, AS subjects demonstrated significantly less preference for social scenes than geometric shapes. Additionally, AS subjects showed less pupil dilation, compared to TDC, when viewing social scenes versus geometric shapes. There was no statistically significant difference found between AS and ASD subjects in either social eye tracking or pupillometry. CONCLUSIONS AND IMPLICATIONS: The use of eye tracking and pupillometry may represent an innovative measure for quantifying AS-associated impairments in social salience.
Authors: Craig A Erickson; Logan K Wink; Bayon Baindu; Balmiki Ray; Tori L Schaefer; Ernest V Pedapati; Debomoy K Lahiri Journal: Am J Med Genet A Date: 2016-06-21 Impact factor: 2.802
Authors: Leigh Sepeta; Naotsugu Tsuchiya; Mari S Davies; Marian Sigman; Susan Y Bookheimer; Mirella Dapretto Journal: J Neurodev Disord Date: 2012-06-07 Impact factor: 4.025
Authors: Molly Winston; Kritika Nayar; Abigail L Hogan; Jamie Barstein; Chelsea La Valle; Kevin Sharp; Elizabeth Berry-Kravis; Molly Losh Journal: Physiol Behav Date: 2019-11-22
Authors: Michael P Hong; Eleanor M Eckert; Ernest V Pedapati; Rebecca C Shaffer; Kelli C Dominick; Logan K Wink; John A Sweeney; Craig A Erickson Journal: J Neurodev Disord Date: 2019-01-21 Impact factor: 4.025